2. Academic Validation
  3. C2orf62 and TTC17 are involved in actin organization and ciliogenesis in zebrafish and human

C2orf62 and TTC17 are involved in actin organization and ciliogenesis in zebrafish and human

  • PLoS One. 2014 Jan 27;9(1):e86476. doi: 10.1371/journal.pone.0086476.
Franck Bontems 1,  Richard J Fish 2,  Irene Borlat 1,  Frédérique Lembo 3,  Sophie Chocu 4,  Frédéric Chalmel 4,  Jean-Paul Borg 3,  Charles Pineau 4,  Marguerite Neerman-Arbez 2,  Amos Bairoch 5,  Lydie Lane 5
Affiliations

Affiliations

  • 1 Department of Human Protein Sciences, Faculty of Medicine, University of Geneva, Geneva, Switzerland.
  • 2 Department of Genetic Medicine and Development, Faculty of Medicine, University of Geneva, Geneva, Switzerland.
  • 3 CRCM - Inserm U1068, Marseille, France ; Institut Paoli-Calmettes, Marseille, France ; CNRS UMR7258, Marseille, France ; Aix-Marseille University, Marseille, France.
  • 4 IRSET - Inserm U1085, Rennes, France.
  • 5 Department of Human Protein Sciences, Faculty of Medicine, University of Geneva, Geneva, Switzerland ; SIB-Swiss Institute of Bioinformatics, Geneva, Switzerland.
PMID: 24475127 DOI: 10.1371/journal.pone.0086476
Abstract

Vertebrate genomes contain around 20,000 protein-encoding genes, of which a large fraction is still not associated with specific functions. A major task in future genomics will thus be to assign physiological roles to all open reading frames revealed by genome sequencing. Here we show that C2orf62, a highly conserved protein with little homology to characterized proteins, is strongly expressed in testis in zebrafish and mammals, and in various types of ciliated cells during zebrafish development. By yeast two hybrid and GST pull-down, C2orf62 was shown to interact with TTC17, another uncharacterized protein. Depletion of either C2orf62 or TTC17 in human ciliated cells interferes with actin polymerization and reduces the number of primary cilia without changing their length. Zebrafish embryos injected with morpholinos against C2orf62 or TTC17, or with mRNA coding for the C2orf62 C-terminal part containing a RII dimerization/docking (R2D2) - like domain show morphological defects consistent with imperfect ciliogenesis. We provide here the first evidence for a C2orf62-TTC17 axis that would regulate actin polymerization and ciliogenesis.

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